Intracranial hypotension headache may occur after dural puncture (postdural puncture headache) or spontaneously (spontaneous intracranial hypotension). The pathophysiology is thought to involve a relative deficit of cerebrospinal fluid (CSF) caused by a leak from the subarachnoid space at a rate greater than CSF production. This deficit causes slight posture-induced shifts in CSF and intracranial contents.1 Downward traction on intracranial structures and increased transmural vascular gradients (cerebral vasodilation) activate stretch pain receptors and cause a characteristic postural headache, which can be incapacitating. Magnetic resonance imaging findings supporting this mechanism include pachymeningeal enhancement (surface vasodilation), engorgement of venous sinuses, and vascular enlargement of the pituitary gland.2 Cranial nerve (CN) deficits can accompany postdural puncture headache (PDPH), and are thought to relate to traction on the nerve itself caused by low CSF volume.3 The most common CNs involved are CN VI (abducens palsy), VII (facial motor deficit), and VIII (hypoacusis).4
In the following, we report the onset of bilateral upper and lower dental pain coincidental with PDPH after combined spinal-epidural (CSE) for cesarean delivery, both of which resolved immediately after an epidural blood patch (EBP). The patient’s initial presentation and subsequent pain are consistent with atypical odontalgia, possibly caused by bilateral traction on CN V2 and V3 secondary to intracranial hypotension after dural puncture.
Written informed consent was obtained from our patient for publication of this report. A copy of this manuscript was sent to her before submission for publication to ensure accuracy.
A 37-year-old woman, gravida 5 para 4, presented for an elective repeat cesarean delivery and postpartum tubal ligation, which were performed under CSE. The epidural space was identified via midline approach at L3–L4 by loss of resistance at a depth of 5 cm using an 18-G Tuohy needle. There was no evidence of dural puncture by the Tuohy needle. After insertion of a 27-G Pencan spinal needle, clear CSF was noted and spinal anesthesia was administered (12.5 mg 0.75% bupivacaine in 8.25% dextrose, 30 µg clonidine, and 0.05 mg hydromorphone). An epidural catheter was then placed and secured at 11.5 cm at the skin. Aside from 2 mL 1.5% lidocaine with 5 µg/mL epinephrine (test dose), no medications were administered through the catheter, which was removed at the conclusion of surgery. Cesarean delivery was uneventful, and the patient was discharged home 2 days later after an uncomplicated hospital course.
On postoperative day (POD) 3, the patient noticed the onset of a mild postural, circumferential, band-like headache. Given the delay in headache onset and a history of dental, sinus, and migraine headache, which never lasted more than a day, she did not consider the possibility of spinal headache. On POD 6, she discussed her persisting headache with a visiting friend, who had been successfully treated with an EBP for PDPH after neuraxial labor analgesia, and who urged her to seek the same treatment. However, our patient delayed medical attention for 2 more days because she was skeptical of the anesthetic technique as the cause of her symptoms, was reluctant to leave her newborn and 4 other children at home to go to the hospital during the winter time, and wanted to avoid unnecessary exposure to hospital pathogens. On POD 8, she returned to the hospital for evaluation of her headache. PDPH was presumed because of its postural nature and after consideration of other causes, including tension headache, migraine, preeclampsia, and cortical venous thrombosis. EBP was proposed and performed in standard fashion. The epidural space was identified with a single midline pass at L3–L4 using an 18-G Tuohy needle. Simultaneously, 16 mL of autologous blood were drawn using aseptic technique and then injected into the epidural space. On conclusion of injection, she stated that her headache had resolved completely. The patient then added, with some surprise, that her toothache was gone, which prompted us to ask, “What toothache?” On further questioning, the patient provided details of dental symptoms that she had noticed the day after onset of her headache, but had not mentioned them because she assumed they were unrelated. She described constant, dull, aching pain in her upper and lower teeth bilaterally. Unlike her headache, her dental pain did not seem to be relieved by recumbent positioning. She had assumed that the pain was related to dental caries that had been diagnosed in early pregnancy, for which she had chosen to postpone treatment until after delivery.
The patient was discharged home 2 hours after the EBP, and the obstetric anesthesia team conducted phone call follow-up over the next 2 days, confirming complete resolution of both headache and dental pain.
Simultaneous development of a severe postural headache accompanied by bilateral upper and lower dental pain within days of CSE anesthesia for cesarean delivery, and their immediate resolution during administration of EBP, suggests intracranial hypotension as a common etiology. The localization of the dental pain corresponded to an area innervated by CN V2 and V3. Our patient’s concurrent dental symptoms, although troublesome to her, were neither solicited nor detected by us until after EBP administration. Like many patients who experience referred neuropathic orofacial pain, she assumed that the dental pain had an odontogenic cause,5 which she intended to have addressed by a dentist. Simultaneous bilateral trigeminal pain is uncommon. In a series of 269 consecutive patients with trigeminal neuralgia, 32 had bilateral symptoms occurring sequentially, and second and third divisions distribution predominated (94%).6 Only 2 patients experienced simultaneous bilateral trigeminal neuralgia, both with central underlying mechanisms—1 with a cerebellopontine angle tumor, the other with hydrocephalus.
Postpartum CN V symptoms have rarely been reported. Fang et al7 described trigeminal and facial nerve palsies, without headache, in a 29-year-old woman after CSE for emergent cesarean delivery. That patient underwent a 6-week course of prednisone and physical therapy, and experienced a near complete return of function of both CNs. Another case8 involved a 38-year-old woman in preterm labor who delivered under epidural analgesia. Three days later, she developed PDPH, right facial numbness (POD 4), and unilateral facial nerve palsy (POD 6). Lacking evidence of dural puncture, an EBP was not offered, and her neurologic symptoms resolved spontaneously by POD 17. There is a single report of a 56-year-old man who experienced paroxysmal, brief, excruciating nonpostural, left unilateral pain in the distribution of CN V2, which developed after many months of untreated, chronic postural headaches after vigorous coughing during an upper respiratory infection.9 Magnetic resonance imaging revealed vertebral artery compression of the inferolateral pons near the left trigeminal nerve root, diffuse pachymeningeal enhancement, cerebellar tonsillar ptosis, and evidence of CSF leak at C8 on the left, which was confirmed by radionuclide cisternography. The symptoms resolved after surgical repair of a C8 nerve root sleeve leak. The authors postulated downward shift of the posterior fossa and vascular compression as the cause of the symptoms.
Our patient’s dental pain was accompanied by severe postural headache and was not limited to a single tooth, but involved her entire mouth. She described the pain as constant, dull, and aching, which is characteristic of atypical odontalgia and neuropathic pain (and distinct from trigeminal neuralgia).5,10 It is not surprising that the recumbent position alleviated the headache but not the dental pain because similar persistence of CN symptoms has been reported, and the mechanisms underlying postural headache (ie, cerebral vasodilation) and intracranial hypotension-induced CN effects may differ.11,12 Traction on CNs caused by caudal shifts of the brainstem (as often observed with imaging studies2,13) is a likely explanation of CN deficits associated with intracranial hypotension headache, in general, and of our patient’s symptoms, in particular. The more acute angulation of CN V2 and V3 compared to V1 as they exit Meckel’s cave may explain the distribution of symptoms. On the other hand, symptoms in the CN V1 distribution may have been present, but clinically indistinguishable from the circumferential headache pain. Although the exact etiology of the observed pain pattern cannot be established with certainty, the complete resolution of both dental pain and severe headache immediately after EBP was striking and suggestive of intracranial hypotension having contributed to her dental pain.
We are unaware of any report of trigeminal nerve pain successfully treated with EBP. The diagnostic classification of atypical odontalgia continues to evolve and its pathophysiology remains unclear.14 However, atypical odontalgia is considered a neuropathic syndrome (referred to as “phantom toothache”).15 We suspect a similar pathophysiologic explanation in our patient. Function of facial (CN VII), abducens (CN VI), and vestibulocochlear (CN VIII) nerves is routinely examined during evaluation of PDPH because they are the most common CNs demonstrating deficits during PDPH. Our experience suggests that trigeminal nerve deficit should also be considered. We did not think of asking about orofacial pain. This report provides additional information regarding neurologic phenomena associated with intracranial hypotension, and may shed further light on the pathophysiology of atypical odontalgia. E
Name: Alexander E. Zajac, MD.
Contribution: This author helped review the literature and write the article.
Name: Michael G. Richardson, MD.
Contribution: This author helped interview the patient, review the literature, and write the article.
This manuscript was handled by: Hans-Joachim Priebe, MD, FRCA, FCAI.
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